Diffuse Intestinal Ganglioneuromatosis Showing Multiple Large Bowel Ulcers in a Patient with Neurofibromatosis Type 1

被引：14

作者：

Iwamuro, Masaya ^{[1
,2
]}

Omote, Rika ^{[3
]}

Tanaka, Takehiro ^{[4
]}

Sunada, Naruhiko ^{[2
]}

Nada, Takahiro ^{[2
]}

Kondo, Yoshitaka ^{[5
]}

Nose, Soichiro ^{[6
]}

Kawaguchi, Mitsuhiko ^{[7
]}

Otsuka, Fumio ^{[2
]}

Okada, Hiroyuki ^{[1
]}

机构：

[1] Okayama Univ, Grad Sch Med Dent & Pharmaceut Sci, Dept Gastroenterol & Hepatol, Okayama, Japan

[2] Okayama Univ, Grad Sch Med Dent & Pharmaceut Sci, Dept Gen Med, Okayama, Japan

[3] Okayama Univ, Grad Sch Med Dent & Pharmaceut Sci, Dept Pathol, Okayama, Japan

[4] Okayama Univ Hosp, Dept Pathol, Okayama, Japan

[5] Okayama Univ, Grad Sch Med Dent & Pharmaceut Sci, Dept Gastroenterol Surg, Okayama, Japan

[6] Okayama Saiseikai Gen Hosp, Dept Anat Pathol, Okayama, Japan

[7] Kawaguchi Med Clin, Kawaguchi, Saitama, Japan

来源：

INTERNAL MEDICINE | 2017年 / 56卷 / 24期

关键词：

diffuse intestinal ganglioneuromatosis; neurofibromatosis type 1; colorectal ulcers; VONRECKLINGHAUSENS DISEASE; GASTROINTESTINAL-TRACT; MYENTERIC PLEXUS; MEGACOLON; VASCULOPATHY; INVOLVEMENT; COLON;

D O I：

10.2169/internalmedicine.8671-16

中图分类号：

R5 [内科学];

学科分类号：

1002 ; 100201 ;

摘要：

A 67-year-old Japanese man with neurofibromatosis type 1 underwent right hemicolectomy owing to abscess formation around the cecum. A pathological analysis revealed diffuse intestinal ganglioneuromatosis in the cecum and colon. Colonoscopy performed eight months after hemicolectomy revealed multiple ulcers throughout the colon and rectum. The colorectal ulcers failed to respond to conservative treatment and ultimately required surgical resection. Diffuse ganglioneuromatosis was observed again in the resected specimen. This report illustrates a rare manifestation of diffuse intestinal ganglioneuromatosis in a patient with neurofibromatosis type 1.

引用

页码：3287 / 3291

页数：5

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